This patient with severe TS presented an ischemic stroke in the posterior cerebral artery and/or postero-inferior temporal branch of the right medial cerebral artery. A stroke work-up was negative with the exception of a positive lupus anticoagulant repeated on various occasions. Systemic diseases were excluded and the patient satisfied the diagnostic criteria for primary APS [
6]. The stroke manifested visual abnormalities and secondary generalized complex partial seizures. Upon reaching a good control of the seizures the manifestations of the TS improved.
The pathogenesis and pathophysiology of TS are still presently unknown. Abnormalities in both the dopaminergic and, to a lesser extent, serotoninergic neurotransmission within the basal ganglia and the cortico-ganglionic-thalamo-cortical circuitry have been suggested [
2].
Both genetic and non-genetic factors may play a role in the pathogenesis of TS. The presence of certain cytogenetic abnormalities, including chromosomal translocations and inversions, and epigenetic risk factors suggest that certain genetic or chromosomal irregularities may be common to some cases of TS [
2]. There is also evidence for an immunological basis in TS. Anti-basal ganglia antibodies appear to play a role in certain TS-spectrum disorders including the pediatric autoimmune neuropsychiatric disorders associated with streptococcal infection (PANDAS). These antibodies have been found in 65% of patients with atypical movement disorders [
3]. Anti-basal ganglia antibodies are often found in children with Sydenham´s chorea. In this disease, antibodies seem to attack the basal ganglia by a mechanism of molecular mimicry triggered by a previous streptococcal infection [
8]. In TS there are higher concentrations of anti-putaminal antibodies compared to normal controls [
5] and immunomodulatory treatment is effective in some patients. There are therapies that may act on a putative immune-mediated attack directed against the nigrostriatal or prefrontal dopaminergic pathways [
3,
9].
Some studies have approached the possible association between aPLAs and TS. Antiphospholipid antibodies have been identified in children with TS and have led to the speculation that these antibodies might play a pathophysiological and possibly pathogenic role in this condition [
7]. If confirmed, the presence of aPLAs has diagnostic and therapeutic implications in TS. However, aPLAs in TS may represent an epiphenomenon rather than a pathophysiological mechanism [
8]. Unfortunately, the number studies on this aspect are scarce and have not included patients with APS.
To our knowledge, this is the first report of a TS patient presenting a stroke in association with APS. Acknowledging a possible chance association, in some TS cases immune phenomena may be involved in the movement disorders. The presence of a particular genetic load or predisposition may increase susceptibility in developing specific antibodies and thrombotic phenomena.
Also of interest in this case is the favorable response of both the epileptic syndrome and the tic-disorder to levetiracetam. It is possible that the seizure disorder may have aggravated the frequency and severity of the tics but we do not exclude a direct effect of levetiracetam on the favorable outcome of the tic-disorder [
10].
In conclusion, this report indicates that aPLAs may be present in some patients with TS. We believe that the mechanisms that trigger the generation of these antibodies leading to vascular events and possibly movement disorders should be further investigated.